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A systematic review of the effectiveness of aerobic exercise interventions for children with cerebral palsy An AACPDM evidence report Written by: Anna Rogers MSc(PT)*. Barbara-Lynne Furler MSc(PT). Stephen Brinks MSc(PT). Johanna Darrah PhD. Approved by AACPDM Treatment Outcomes Committee Review Panel: American Academy for Cerebral Palsy and Developmental Medicine Lisa Samson-Fang, MD Johanna Darrah, PhD John McLaughlin, MD Lynne Logan, MA, PT William Walker MD Lesly Wiart, MScPT Meg Barry-Michaels PhD, PT, PCS Michael Msall MD Unni Narayanan MD Laura Vogtle, PhD, PT Robbin Hickman PT MHS PCS Alexander Hoon, MD Corresponding Author Anna Rogers 6227 109A St. Edmonton, AB T6H 3C6 Canada Tel: (780) 862-9244 Email:[email protected]

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A systematic review of the effectiveness of aerobic exercise interventions for children

with cerebral palsy

An AACPDM evidence report

Written by:

Anna Rogers MSc(PT)*. Barbara-Lynne Furler MSc(PT). Stephen Brinks MSc(PT).

Johanna Darrah PhD.

Approved by AACPDM Treatment Outcomes Committee Review Panel: American Academy for

Cerebral Palsy and Developmental Medicine Lisa Samson-Fang, MD Johanna Darrah, PhD John McLaughlin, MD Lynne Logan, MA, PT William Walker MD

Lesly Wiart, MScPT Meg Barry-Michaels PhD, PT, PCS Michael Msall MD Unni Narayanan MD

Laura Vogtle, PhD, PT Robbin Hickman PT MHS PCS Alexander Hoon, MD

Corresponding Author Anna Rogers 6227 109A St. Edmonton, AB T6H 3C6 Canada Tel: (780) 862-9244 Email:[email protected]

Aerobic Exercise Systematic Review 1

Abstract

The aim of this review was to assess the evidence regarding the effectiveness of aerobic training

interventions for children with cerebral palsy. The target population included children with

cerebral palsy of any severity, aged 2 to 17 years old. The following databases were searched for

English studies from 1960 to the present: MEDLINE, EMBASE, CINAHL, Pascal, Cochrane

Library, CSA Neuroscience Abstracts, PEDro and Sport Discus Search terms included cerebral

palsy, athetoid, ataxic, spastic diplegia, hemiplegia, quadriplegia, aerobic, exercise, training,

physical activity, aquatic/water/pool therapy, and continuous exercise. The American Academy

of Cerebral Palsy and Developmental Medicine (AACPDM) systematic review guidelines were

used to format the review. One thousand four hundred and eighty nine articles were identified

and examined for the stated inclusion and exclusion criteria. Thirteen articles met the criteria for

inclusion. The evidence suggests that aerobic exercise with children with cerebral palsy can

improve physiological outcomes, but the influence of these changes on outcomes representing

activity and participation in unknown. Future research needs improved methodological rigour in

order to determine a specific set of exercise guidelines and safety considerations.

Aerobic Exercise Systematic Review 2

The AACPDM has undertaken the development of systematic reviews to summarize the

literature about specific intervention strategies used to assist children with developmental

disabilities. These reviews are not best practice documents or practice guidelines, but rather they

gather and present the best evidence – for and against – the effectiveness of an intervention.

Their goal is to present the evidence about interventions in an organized fashion to identify gaps

in evidence and help address new research that is needed. The Academy is neither endorsing nor

disapproving of an intervention in these reviews. Every effort has been made to assure that

AACPDM systematic reviews are free from real or perceived bias. Details of the disclosure and

consensus process for AACPDM outcomes reports can be viewed at www.AACPDM.org.

Nevertheless, the data in an AACPDM Systematic Review can be interpreted differently,

depending on people's perspectives. Please consider the conclusions presented carefully.

Background

Therapeutic programs for adults and children with disabilities are increasingly

incorporating a fitness component into intervention strategies. The benefits of aerobic exercise

for persons with disabilities include increased cardiovascular capacity and endurance, weight

management and lower blood lipid levels, preservation of bone mass and overall maintenance of

function.2 In addition, the opportunity for persons with disabilities to participate in community

aerobic fitness programs allows them to take responsibility for their own health and fitness.

People with a physical disability have identified numerous barriers to participation in physical

activities including physical accessibility to exercise facilities, shortage of adapted exercise

equipment, inability to pay for a fitness membership and a lack of knowledge in the fitness and

exercise professions about how to appropriately design an exercise program for specific

Aerobic Exercise Systematic Review 3

disabilities.3 Health counseling about the importance of physical activity and its accessibility for

persons with disabilities is minimal.3

Historically, exercise programs that included aerobic or muscle strengthening

components were often contraindicated for persons with cerebral palsy because of the concern

that increased effort and exertion during exercise would result in increased muscle tone, a

decrease in range of motion, and/or an overall decrease in function.4 Concerns about the safety of

progressive resisted muscle strengthening for children with cerebral palsy have been negated by

recent studies that demonstrate no increase in spasticity and no loss of range of motion following

a resistance training program.4-6 While strength training is now recognized as an effective

intervention for improving muscle strength with children with cerebral palsy4, the efficacy of

aerobic exercise as a safe and beneficial intervention option for children with cerebral palsy has

not been reviewed in the literature. The purpose of this review is to review the evidence for the

effectiveness of aerobic exercise interventions for children with cerebral palsy.

Method

OPERATIONAL DEFINITION AND INCLUSION CRITERIA

Aerobic exercise was defined as any activity that increases heart rate or proposes to

increase heart rate to exact physiological change. The outcomes evaluated represented more than

just physiological measures such as 6 minute walk distance or gait efficiency. All outcomes

included in the studies were documented. Studies with a sample that included children aged 2 to

17 years with any severity of cerebral palsy diagnosis were eligible for inclusion. At least 50%

of subjects in the study sample had to have a diagnosis of cerebral palsy, or the results of the

subjects with cerebral palsy must be distinguishable from children with other diagnoses and

analyzed separately.

Aerobic Exercise Systematic Review 4

LITERATURE SEARCH

A medical librarian assisted with the search strategy. The following databases were

searched in English only from 1960 to 2006: CINAHL (297 studies), Cochrane Library (65),

CSA Neuroscience Abstracts (1), EMBASE (464), MEDLINE (372), Pascal (64), The

Physiotherapy Evidence Database (PEDro) (3), and Sport Discus (223). The search terms used

for the target population were: cerebral palsy, athetoid, ataxic, spastic diplegia, and quadriplegia.

The intervention search terms were: aerobic, exercise, training, physical activity,

aquatic/water/pool therapy, and continuous exercise. A specific pediatric search, ARCHE

(Alberta Research Centre Health Evidence), was also applied to the OVID search engines. The

initial search yielded 1489 articles; 1205 articles were excluded based on the inappropriateness

of their titles. The remaining 284 abstracts were read independently by two reviewers and

conflicts were resolved by a third reader if required. Two-hundred seventy-one articles were

excluded for one or more of the following reasons: 1) lack of aerobic exercise intervention, 2) no

reported aerobic fitness outcomes, 3) use of a one-time exercise protocol, 4) the population did

not match the inclusion criteria, or 5) review articles. Thirteen articles met the inclusion criteria.

ORGANIZATION OF EVIDENCE

Outcome measures were classified according to the components of the International

Classification of Functioning, Disability and Health (ICF)7 as presented in Table I. Studies were

classified using a hierarchy of research design rigour (Table II). Level I studies produce results

from which definitive conclusions can be made. Level II thru level IV studies report

progressively less credible evidence from which only cautious conclusions can be drawn. No

definite conclusions can be made from level V evidence.

Aerobic Exercise Systematic Review 5

Table III summarizes the 13 studies reviewed. Two small, randomized controlled trials

had control groups that received the same intervention as the experimental group but at a reduced

frequency.8,9 One cohort study used the subjects that dropped out or did not begin training as the

control group.10 Five studies compared the results of a pre-test/post-test in a single group with no

control using a variety of aerobic exercise programs ranging from games to bicycle ergometry.11-

15 The remaining studies consisted of 2 case studies 16,17 and 3 single subject design studies.18-20

Tables IV and V include only evidence from the studies that received a level of evidence

rating of level III or higher. Level IV and V studies are not included in the final evidence tables

because of threats to internal validity due to weak study design. In addition, the level of evidence

of single subject design studies cannot be rated within the levels defined for group studies. Table

IV provides the conduct ratings of the studies rated as level III evidence and higher. The conduct

rating provides an assessment of the quality of the study based on the score from the seven

evaluative questions under Table IV. Each study was rated by two independent reviewers and a

third reader was available to resolve conflict and determine the final rating if necessary.

Table V summarizes the outcomes of interest, the measures used to quantify the

outcomes, the ICF components of health represented by the outcome measures used, and the

statistical results. No study reported power calculations and thus studies reporting a ‘not

reported’ (nr) or ‘not significant’ (ns) result may not have enough power to detect true

differences between groups.

Analysis and discussion of the evidence

1. WHAT EVIDENCE EXISTS ABOUT THE EFFECTS OF AEROBIC EXERCISE ON

MEASURES REPRESENTING THE COMPONENTS OF BODY FUNCTION AND

STRUCTURE?

Aerobic Exercise Systematic Review 6

All outcomes measured in the studies reported in Tables IV and V represent the

components of body structure and function. All three studies evaluated physiological variables

related to aerobic fitness, two reported body composition9,10 and one study measured grip

strength and mechanical efficiency10 but these two outcomes were not analyzed statistically.

Aerobic fitness.

Significant improvement in aerobic fitness following training was reported in all three

studies. Different outcome measures were used to determine change in aerobic fitness, including

heart rate (HR) during sub-maximal and maximal tests,10 VO2peak and VO2max9,10 and the

relationship between VO2 and workload and VO2 and heart rate.8

The level II study by van den Berg-Emons et al. demonstrated an improvement in aerobic

fitness following two, 9-month, “high intensity” training sessions 45 minutes in duration, four

times a week which involved cycling, wheelchair skills, running, swimming and mat exercises.9

In contrast, Dresen et al. (level II) showed a significant decrease in VO2/workload after a 10-

week training program that consisted of regular physical education activities (judo, swimming

and games) for a total of two hours per week.8 The only difference between the control and

experimental groups in this study was that the latter simply received undefined “active

encouragement” from the instructor. Van den Berg-Emons et al. found a significant increase in

peak aerobic power after training.9 This same increase was not observed following a two month

rest period, suggesting that although children with cerebral palsy are able to improve their level

of aerobic fitness, it may not be maintained over time if the exercise level is not maintained.

Body composition.

Both van den Berg-Emons et al.9 (level II) and Bar-Or et al.10 (level III) used the

measures of height, weight and skinfolds to report anthropometric variables. Bar-Or et al.10

Aerobic Exercise Systematic Review 7

reported significant change in the height variable, in both the control and exercise groups, while

van den Berg-Emons et al.9 measured fat mass (skinfolds) and showed a significant increase in

the control group.

2. WHAT EVIDENCE EXISTS ABOUT THE EFFECTS OF AEROBIC EXERCISE ON

MEASURES REPRESENTING THE COMPONENTS OF ACTIVITY AND

PARTICIPATION?

None of the studies in the evidence tables reported outcomes representing the ICF

components of activity and participation.

3. WHAT KINDS AND MAGNITUDE OF COMPLICATIONS HAVE BEEN

DOCUMENTED?

Safety concerns were not discussed in any articles, including studies categorized as Level

IV and V. There were no reports of increased spasticity, medical problems or the occurrence of

musculoskeletal trauma in any of the studies reviewed. Evaluation of adverse effects such as

fatigue was not reported.

4. WHAT IS THE STRENGTH OF THE EVIDENCE?

The results of the three level II-III studies all suggest that aerobic exercise intervention

can improve physiological outcomes in children with cerebral palsy. However, the total number

of subjects in the review is only 58, and the largest single sample was 26 participants.10 As

described in Table III, most samples were extremely variable both in the ages of the children and

their physical abilities. Given the small sample size, the heterogeneity of both the samples and

inventions and the poor conduct ratings received by the Level II and III studies (Table IV), it is

difficult to identify specific components of aerobic intervention that are most effective.

Aerobic Exercise Systematic Review 8

The age range of participants among the studies ranged from 7 to 22 years. The physical

abilities of the children within the same study ranged from mild to severe diplegia and

hemiplegia, and ability levels included ambulatory and non-ambulatory. This heterogeneity of

age and motor abilities makes it difficult to discern the extent to which each subject was able to

participate in an exercise program. Children with differing motor abilities and different ages may

respond differently to aerobic exercise intervention. The samples in the studies were all too

small to allow for subgroup analyses.

Most of the interventions are non-replicable because of lack of detail provided.

Intervention activities ranged from game activities in a physical education setting with “active

encouragement”8 to more specific aerobic exercise such as cycling, wheelchair skills and

running. There was no description as to whether the activity was continuous or non-continuous

or what equipment/intervention adaptations had to be made for the non-ambulatory participants.

Exercise parameters varied among the studies; intensity of exercise varied from inconsistent

monitoring of 160 beats per minute8 to subjective opinions based on investigators’ observations

during game play.10 Frequency varied from a total of 2-6 hours a week spread over a range of 2-

4 exercise sessions a week. Duration of the intervention programs varied from 10 weeks to 18

months.8,9 Standardized guidelines for frequency, duration, intensity and type of exercise

necessary to impose physiological change cannot be extracted from this body of literature.

The measures used in the level I to III studies focused on the outcomes of body structure

and function. All three studies used oxygen consumption (VO2), heart rate (HR) or the

relationship between VO2 and HR to detect change in aerobic fitness, but these measures were

not standard across or even within these studies. For example, Bar-Or et al.10 used both a sub-

maximal and a maximal VO2 test, whereas Dresen et al.8 used only sub-maximal VO2 testing.

Aerobic Exercise Systematic Review 9

Although both groups of authors reported an improvement in oxygen consumption, they are

reporting different outcomes (VO2max vs. sub-maximal VO2 to workload ratios) and therefore do

not allow for the discussion of a trend across studies. No studies evaluated the relationship

between improvement of aerobic fitness at a physiological level and improvement of outcomes

representing the ICF components of activity and participation. Therapists are interested in

optimizing the functional abilities of children with cerebral palsy and future studies need to

systematically evaluate the relationship of outcomes representing components of body function

and structure and activity and participation. For example, does an increase in VO2max result in

improved walking endurance or mobility in school?

Summary and directions for future research.

The limited evidence available suggests that children with cerebral palsy who participate

in an aerobic exercise training program demonstrate improvements in physiological measures of

aerobic fitness. However the body of research has methodological limitations and is based on a

small sample size.

The current emphasis on functional goals and community exercise opportunities for

children with cerebral palsy may have moved research about the effects of aerobic exercise too

rapidly from controlled laboratory settings to community exercise programs. The paucity of

controlled laboratory research suggests that more information is needed from laboratory settings

to establish the efficacy of aerobic exercise models using exercise regimes with different

intensity, frequency and duration parameters. When these parameters are better established and

understood, then the effectiveness of the programs in community setting could be systematically

evaluated. However, because of the interest in community fitness programs for children with

cerebral palsy, it is doubtful that the community programs will wait until the efficacy

Aerobic Exercise Systematic Review 10

information from laboratory settings is available. If the research in the two different settings

continues to develop simultaneously, it would be valuable to collect similar information from

both settings for comparison. By using the same cadre of measures representing all components

of the ICF in both controlled laboratory condition and community settings, future research

endeavors can begin to systematically evaluate the relationships across the ICF components.

Although the study designs of levels IV and V evidence inherently limit the confidence

that can be placed in these results, it is interesting to note that measures that target outcomes

within the ICF components of activity and participation were used in some of these studies.

Several studies reported an improvement in gait efficiency 16,17 and in the distance walked during

a 6-minute walk test 19. A few authors reported improvement in psychological variables such as

self-perception of physical appearance 11, athletic competence and self-worth using either the

Self Perception Profile for Adolescents (SPPA) or College students (SPPC).12,20 These types of

outcomes need to be incorporated in studies of more rigorous research design. No complications

or safety concerns were reported in any of the Level IV and V studies.

The finding that children with cerebral palsy are able to train to elicit physiological

change is a positive outcome that should serve as a catalyst for future well-designed studies to

establish safe, effective, and specific exercise guidelines for children of different ages and ability

levels.

Acknowledgements: Dr. Darrah was funded as a CIHR New Investigator during the preparation

of this review. Michelle Kelly MScPT reviewed the manuscript and provided valuable

suggestions.

11

References

1. O’Donnell M, Darrah J, Adams R, Butler C, Roxborough L, & Damiano D. (2004)

AACPDM methodology to develop systematic reviews of treatment interventions.

http://www.aacpdm.org/resources/systematicReviewsMethodology.pdf (accessed

November 2005).

2. Rimmer JH. (2005) Exercise and physical activity in persons aging with a disability. Phys

Med Rehabil in North America 16: 41-56.

3. Rimmer, JH, Riley B, Wang E, Rauworth A, & Jurkowski J. (2004) Physical activity

participation among persons with disabilities: barriers and facilitators. Am J Prev Med

26(5): 419-425.

4. Dodd KJ, Taylor NF, & Damiano DL. (2002) A systematic review of the effectiveness of

strength-training programs for people with cerebral palsy. Arch Phys Med Rehabil 83(8):

1157-1164.

5. MacPhail HE, & Kramer JF. (1995) Effect of isokinetic strength-training on functional

ability and walking efficiency in adolescents with cerebral palsy. Dev Med Child Neurol

37: 763-775.

6. Tweedy, S. (1995) Strength training for athletes with cerebral palsy. Ultra-Fit Australia,

26: 66-70.

7. World Health Organization. (2001) International Classification of Functioning,

Disability and Health. Geneva: World Health Organization.

8. Dresen MH, de Groot G, Mesa Menor JR, & Bouman, LN (1985) Aerobic energy

expenditure of handicapped children after training. Arch Phys Med Rehabil 66(5): 302-

306.

12

9. Van den Berg-Emons RJ, Van Baak MA, Speth L, & Saris WH. (1998) Physical training

of school children with spastic cerebral palsy: effects on daily activity, fat mass and

fitness. Int J Rehabil Res 21(2): 179-194.

10. Bar-Or O, Inbar O, & Spira R. (1976) Physiological effects of a sports rehabilitation

program on cerebral palsied and post-poliomyelitic adolescents. Medicine & Science in

Sports 8(3): 157-161.

11. Berg K. (1970) Effect of physical training of school children with cerebral palsy. Acta

Paediatr Scand 204 (Suppl): 27-33.

12. Darrah J, Wessel J, Nearingburg P, & O'Connor M. (1999) Evaluation of a community

fitness program for adolescents with cerebral palsy. Pediatr Phys Ther 11(1): 18-23.

13. Lundberg A, Ovenfors CO, & Saltin B. (1967) Effect of physical training on school-

children with cerebral palsy. Acta Paediatr Scand 56(2): 182-188.

14. Lundberg A, & Pernow B. (1970) The effect of physical training on oxygen utilization

and lactate formation in the exercising muscle of adolescents with motor handicaps.

Scandinavian Journal of Clinical & Laboratory Investigation 26(1): 89-96.

15. Shinohara TA, Suzuki N, Oba M, Kawasumi M, Kimizuka M, & Mita K. (2002) Effect

of exercise at the AT point for children with cerebral palsy. Bulletin of the Hospital for

Joint Diseases, 61(1-2), 63-67.

16. Wiepert SL, & Lewis CL. (1998) Effects of a 6-week progressive exercise program on a

child with right hemiparesis. Physical Therapy Case Reports, 1(1), 21-26.

17. Fragala-Pinkman MA, Haley SM, Rabin J, & Kharasch VS. (2005) A fitness program for

children with disabilities. Physical Therapy, 85(11), 1182-1200.

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18. Rintala P, Lyytinen H, & Dunn JM. (1990) Influence of a physical activity program on

children with cerebral palsy: A single subject design. Pediatr Exerc Sci 2(1): 46-56.

19. Mulligan H, Abbott S, Clayton S, McKegg P, & Rae R. (2004) The outcome of a

functional exercise programme in an adolescent with cerebral palsy: A single case study.

New Zealand Journal of Physiotherapy, 32(1), 30-38.

20. Schlough K, Nawoczenski D, Case LE, Nolan K, & Wigglesworth JK. (2005) The effects

of aerobic exercise on endurance, strength, function and self-perception in adolescents

with spastic cerebral palsy: A report of three case studies. Pediatr Phys Ther 17(4): 234-

250.

14

Table I

Classification of Outcomes ICF Component Definition Body Function Body functions are the physiological

functions of body systems including psychosocial functions

Body Structure Body structures are anatomical parts of the body such as organs, limbs, and their components

Activity Activity is the execution of a task or action by an individual

Participation Participation is involvement in a life situation

Context/Environmental Factors Environmental factors make up the physical, social and attitudinal environment in which people live and conduct their lives

15

Table II

Levels of Evidence

Level Intervention (Group) studies

I

Systematic Review of randomized controlled trials (RCTs) Large RCTs (with narrow confidence intervals) (n >100)

II

Smaller RCTs (with wider confidence intervals) (n <100) Systematic Reviews of cohort studies “Outcomes research” (very large ecologic studies)

III Cohort studies (must have concurrent control group) Systematic Reviews of Case Control Studies

IV

Case series Cohort study without concurrent control group (e.g. with historical control group) Case-control study

V

Expert Opinion Case Study or report Bench research Expert opinion based on theory or physiologic research Common sense/anecdotes

16

Table III Summary of Studies: Interventions and Participants

Study Level of

Evidence/ Research Design

Intervention Control Intervention Population Total n Ages

1967 Lundberg et al. IV Pre-test/ Post-test with no control group

Continuous large muscle group exercise (running, jumping, parallel bars): 30 min, 2x/wk Duration: 6 wk

None CP: 3 hemiplegia, 3 diplegia, 3 tetraplegia, 4 athetosis, 1 ataxia

14 15-20y

1970 Berg IV Pre-test/ Post-test with no control group

Bicycle ergometer at varying intensities: 20 min, 3x/wk Duration: 1.5- 16 mo

None CP: 12 spastic diplegia or tetraplegia, 10 dyskinetic disturbances

22 7-25y

1970 Lundberg & Pernow

IV Pre-test/Post-test with no control group

Continuous large muscle exercise (running, jumping, parallel bars, dumbbells, medicine balls, pulleys): 30 min, 2x/wk Duration: 6 wk

None CP: 3 diplegia, 3 athetosis, 1 hemiplegia, 1 ataxia, 1 post-traumatic spastic hemiplegia

9 15-25y

1976 Bar-Or et al. III-W (1/7) Cohort Study

Combination of group and individual games

9 children with cerebral palsy that did not begin or

CP. 26 Spastic diplegia with 2 subjects with a mix of

26 15-22y

17

(table tennis, shuffleboard, bowling) and mat exercises: 2hr/d, 2d/wk Duration: 12 mo

stopped training within first 2 mo

spasticity and athetosis. All ambulatory to varying degrees

1985 Dresen et al. II-W (3/7) Small RCT

Regular physical education classes (judo, swimming and games): 3x/wk for total of 2hr/wk Duration: 10 wk

As intervention without active encouragement.

CP: 6 spastic hemiplegia, 3 spastic diplegia, 1 dwarf growth, 1 cerebellar ataxia. 4 of 11 were being treated for epilepsy

12; EXP= 6 CON=5 1 drop-out

8-14y

1990 Rintala et al. Single Subject Design

Rotation through activities targeting static and dynamic balance, physical fitness and catching and throwing skills: 1hr/d, 2 d/wk for a total of 30 sessions. Duration: 4 mo

None CP: 8 with either hemiplegia or diplegia, All ambulatory

8 7-11y

1998 van den Berg- Emons et al.

II-W (2/7) Small RCT

a) First year Intervention: Group aerobic exercise program (cycling, wheelchair skills, running, swimming, flying saucer, mat exercises): 45 min,

Regular school physical education and individual physiotherapy sessions.

CP: 16 diplegia, 4 tetraplegia. 10 ambulatory, 10 used wheelchair

20 a) EXP= 10 CON=10 b) EXP=17 CON=10

7-13y

18

4x/wk in addition to regular PE and PT Duration: 9 mo b) Second Year Intervention: As in a) minus swimming and isokinetic strengthening: 45 min, 2x/wk plus regular PE and PT Duration: 9 mo

1998 Wiepert & Lewis

V Case Study PT led activities including ball play, step-ups, prolonged walking, tricycle riding: 1hr/d, 2d/wk Duration: 6 wk

None 1 child with hemiparetic CP

1 3y

1999 Darrah et al. IV Pre-test/Post-test with no control group

Circuit training program including aerobics, strengthening, flexibility: 70-90min, 3x/wk Duration: 10 wk

None CP: 13 hemiplegia, 5 diplegia, 2 quadraplegia, 2 ataxia, 1 dystonia. 2 non-ambulatory, 1 used a walking stick, 20 independent ambulators

23 11-20y

2002 Shinohara et al. IV Pre-test/ Post-test with no control group

Children trained at anaerobic threshold (AT) point on either arm or leg cycle ergometer (as able):

None 11 CP. All ambulatory to varying degrees.

11 13.3- 15.8y

19

20 min, 1.8 d/wk Duration: 6 to 20 wk

2004 Mulligan et al. Single Subject Design

Cardiovascular and strength conditioning program at outpatient facility: 1d/wk, 3 additional strength sessions at home and encouragement to increase daily physical activity Duration: 6 wk

None CP: 1 spastic diplegia 1 13y

2005 Fragala-Pinkham et al.

IV Case Report a) Group fitness program (movement to music, parachute games, obstacle courses, games, riding bicycles, strength training): 60-70min, 2x/wk Duration: 14 wk b) Non-simultaneous home exercise program: written instructions and video taped aerobics and

None CP: 1 hemiplegia, 3 spastic diplegia, Non- CP: 1 anoxic brain injury, 2 developmental coordination disorders, 1 pervasive developmental disorder, 1 developmental delay

9 5-9y

20

strengthening activities: 2x/wk Duration: 3 mo

2005 Schlough et al. Single Subject design

Cardiovascular exercise machines: treadmill, elliptical, recumbent stepper: 2-5min/d which was progressed, 3x/wk Duration: 6 wk

None CP: 3 with either spastic hemiplegia or diplegia. All ambulatory

3 17-20y

min-minute; wk-week(s); CP-cerebral palsy; y-year(s); mo-month(s); hr-hour(s); d-day(s); EXP-experimental; CON-control; PE-physical education; PT-physical therapy

21

Table IV

Conduct of Study

Study Level/Quality 1 2 3 4 5 6 7 1976 Bar-Or et al. III strong Yes Yes No Yes Yes Yes Yes 1985 Dresen et al. IImoderate No Yes No Yes Yes No Yes 1998 van den Berg- Emons et al.

IImoderate Yes Yes No Yes Yes No Yes

Conduct of a study was rated as Strong (score 7 or 6), Moderate (score 5 or 4), or Weak (score <3) 1. Were inclusion and exclusion criteria of the study population well described and followed? 2. Was the intervention well described and was there adherence to the intervention assignment? For 2-group designs was the control exposure also well described? 3. Were the measures used clearly described, valid and reliable for measuring the outcomes of interest? 4. Was the outcome assessor unaware of the intervention status of the participants (i.e. were there blind assessments)? 5. Did the authors conduct and report appropriate statistical evaluation including power calculations? 6. Were dropouts/loss to follow-up reported and less than 20%? For 2-group designs, were dropouts balanced? 7. Considering the potential within the study design, were appropriate methods for controlling confounding variables and limiting potential biases used?

22

Table V

Summary of Studies: Outcomes, Measures, and Results

Study Outcome of Interest Measure Components of Health Body

Structure(s)/Function Activities & Participation

Contextual Factors

1976 Bar-Or et al. Body Composition Aerobic Fitness Grip Strength Mechanical Efficiency Submaximal Heart Rate

Height Weight Skinfolds Submaximal test Maximal test (peak VO2) Handheld dynamometer Submaximal test Bipolar ECG

nr nr nr p<0.01 EXP (CP) p<0.05 EXP (CP) nr nr p<0.01 EXP (CP)

1985 Dresen et al. VO2/ WL (POST) VO2/WL (FOLLOW-UP) VO2/ HR Exercise Intensity (HR ≥ 160bpm)

Submaximal test Submaximal test Submaximal test Telemetric transmitters, HR memories, chest cables

p<0.05 EXP p<0.05 EXP ns p<0.05

1998 van den Berg- Emons et al. *

Body Composition Level of Daily Physical Activity (PA) Peak Aerobic Power

Height Weight Skinfolds Fat mass TEE/ SMR and/or TEE/RMR Cycling test

nr nr nr p<0.05 CON (year 1) ns (year 1), p<0.05 EXP (year 2) p<0.01 EXP (year 1), p<0.01 EXP (year 2) but no difference between

23

Peak Anaerobic Power Isokinetic Strength

Cycling test Cybex II

CON and EXP (year 2) ns p<0.05 EXP (year 2) for most affected leg

VO2-oxygen uptake; ECG-electrocardiograph; nr-not reported; EXP-experimental; CP-cerebral palsy; VO2/WL-oxygen uptake to workload ratio; VO2 /HR-oxygen uptake to heart rate ratio; HR-heart rate; bpm-beats per minute; ns-not statistically significant; TEE/SMR-total energy expenditure/ sleeping metabolic rate; TEE/RMR-total energy expenditure/ resting metabolic rate; CON-control *Results: The van den Berg-Emons et al. study was carried out over 2 years. The experimental group in the first year consisted of 10 subjects that participated in 4, 45 min. exercise sessions 4 times a week in addition to regular physical education and individual physical therapy (PT) sessions while the control group solely received the physical education classes and individual PT. At 9 months follow-up testing, physical activity level increased by 16% but was not statistically significant (p<0.07) and was not different from the control group. Fat mass (FM) continued to increase over the 9-month school term in the CON (p<0.05 at 9 months follow-up) with no change in FM in experimental group. Peak aerobic power increased by 35% in EXP and was significantly greater than CON at 9 months (p<0.01). Anaerobic power increased in CON (p<0.05) and a similar trend was demonstrated in EXP (p<0.06) with no difference between groups, suggesting the change may be a result of maturation of the subjects. Following the summer, holidays peak aerobic power decreased significantly in the EXP group by 17% (p<0.01). In the second year of the study, the original EXP group and the original CON group both participated in 2, 45 min exercise sessions a week in addition to physical education class and individual PT and were labeled as EXP2xtot and EXP2x, respectively. The groups were also compared to the original CON. Following the second year of training, there was no significant increase in PA in either EXP2xtot, EXP2x and the change in PA was not different from the change that occurred in CON suggesting that 2 additional exercise sessions/ week did not increase PA level more than the change in CON with only physical education classes and individual PT. Fat mass was significantly lower in the children who trained 4 times a week (EXP) from the first year of study, compared to those who trained only twice a week (EXP2x) (p<0.05). There was a significant increase in peak aerobic power in EXP2x (p<0.01) at 9 months but the difference between EXP2x and CON was not significant. Isokinetic muscle strength increased in the most affected leg in EXP2xtot at 9 months (p<0.05).